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    Rate of familial amyotrophic lateral sclerosis: a systematic review and meta-analysis.

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    Authors
    Byrne, Susan
    Walsh, Cathal
    Lynch, Catherine
    Bede, Peter
    Elamin, Marwa
    Kenna, Kevin
    McLaughlin, Russell
    Hardiman, Orla
    Affiliation
    Deparment of Neurology, Beaumont Hospital, Dublin, Ireland.
    Issue Date
    2010-11-03
    
    Metadata
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    Citation
    Rate of familial amyotrophic lateral sclerosis: a systematic review and meta-analysis. 2010: J. Neurol. Neurosurg. Psychiatr.
    Journal
    Journal of neurology, neurosurgery, and psychiatry
    URI
    http://hdl.handle.net/10147/126131
    DOI
    10.1136/jnnp.2010.224501
    PubMed ID
    21047878
    Abstract
    Background The population rate of familial amyotrophic lateral sclerosis (FALS) is frequently reported as 10%. However, a systematic review and meta-analysis of the true population based frequency of FALS has never been performed. Method A Medline literature review identified all original articles reporting a rate of FALS. Studies were grouped according to the type of data presented and examined for sources of case ascertainment. A systematic review and meta-analysis of reported rates of FALS was then conducted to facilitate comparison between studies and calculate a pooled rate of FALS. Results 38 papers reported a rate of FALS. Thirty-three papers were included in analysis and the rate of FALS for all studies was 4.6% (95% CI 3.9% to 5.5%). Restricting the analysis to prospective population based registry data revealed a rate of 5.1% (95% CI 4.1% to 6.1%). The incidence of FALS was lower in southern Europe. There was no correlation between rate of FALS and reported SOD1 mutation rates. Conclusion The rate of FALS among prospective population based registries is 5.1% (CI 4.1 to 6.1%), and not 10% as is often stated. Further detailed prospective population based studies of familial ALS are required to confirm this rate.
    Item Type
    Article In Press
    ISSN
    1468-330X
    ae974a485f413a2113503eed53cd6c53
    10.1136/jnnp.2010.224501
    Scopus Count
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    Beaumont Hospital

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